en POLSKI
eISSN: 2083-8441
ISSN: 2081-237X
Pediatric Endocrinology Diabetes and Metabolism
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SCImago Journal & Country Rank
 
1/2022
vol. 28
 
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abstract:
Case report

Atypical parathyroid adenoma: Severe manifestations in an adolescent girl

Hiya Boro
1
,
Sarah Alam
1
,
Vijay Kubihal
2
,
Saurav Khatiwada
1
,
Suraj Kubihal
1
,
Shipra Agarwal
3
,
Rajesh Khadgawat
1

1.
Department of Endocrinology and Metabolism, All India Institute of Medical Sciences (AIIMS), New Delhi, India
2.
Department of Radiodiagnosis, All India Institute of Medical Sciences (AIIMS), New Delhi, India
3.
Department of Pathology, All India Institute of Medical Sciences (AIIMS), New Delhi, India
Pediatr Endocrinol Diabetes Metab 2022; 28 (1): 91–100
Online publish date: 2022/02/11
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Introduction
Primary hyperparathyroidism (PHPT) is a disease that is usually diagnosed in an asymptomatic state during routine biochemical screening. It generally manifests as a sporadic disease in post-menopausal women. However, in India and developing countries, we continue to see severe skeletal and renal manifestations of the disease.

Case report
Herein, we describe the case of a 16-year-old adolescent girl who presented with severe manifestations of primary hyperparathyroidism. Biochemically, she had severe parathyroid hormone (PTH)-dependent hypercalcaemia with hypophosphataemia and vitamin D deficiency (serum total Ca – 18.5 mg/dl [8.5–10.5 mg/dl], serum PO4 – 1.9 mg/dl [2.5–4.5 mg/dl], serum ALP – 2015 IU/l [80–240 IU/l], serum 25[OH]D – 19.1 ng/ml [30–100 ng/ml] and serum iPTH > 5000 pg/ml [15–65 pg/ml]). Pre-operatively, she required management with saline diuresis, bisphosphonate, and calcitonin. After surgery, the patient had severe hungry bone syndrome (serum Ca – 4.1 mg/dl, serum PO4 – 2.1 mg/dl, serum ALP > 10,000 IU/l) that required treatment with calcium infusions for almost 3 months. Although the clinical and biochemical picture was suggestive of parathyroid carcinoma, histopathology revealed atypical parathyroid adenoma with low proliferative index. Atypical parathyroid adenoma is a term applied to a neoplasm with ‘worrisome’ features but not fulfilling the ‘absolute histopathological criteria of malignancy’.

Conclusions
Atypical parathyroid adenoma, a rare cause of PHPT, may be associated with severe manifestations. Although malignancy was not discerned in the immediate post-operative period, we plan to continue long-term follow-up of the patient to look for any signs of recurrence or development of parathyroid carcinoma.

keywords:

primary hyperparathyroidism, atypical parathyroid adenoma, parathyroid carcinoma


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